Total submissions: 3
Submitter | RCV | SCV | Clinical significance | Condition | Last evaluated | Review status | Method | Comment |
---|---|---|---|---|---|---|---|---|
Broad Center for Mendelian Genomics, |
RCV001248894 | SCV001422575 | uncertain significance | Maturity-onset diabetes of the young type 3 | 2020-01-22 | criteria provided, single submitter | curation | The p.Glu235Gln variant in HNF1A has been reported in an Indian individual with maturity-onset diabetes of the young (PMID: 19336507), and has been identified in 0.003% (1/30604) of South Asian chromosomes by the Genome Aggregation Database (gnomAD, http://gnomad.broadinstitute.org; dbSNP rs1353807357). Please note that for diseases with clinical variability, or reduced penetrance, pathogenic variants may be present at a low frequency in the general population. In vitro functional studies provide some evidence that the p.Glu235Gln variant may slightly impact protein function (PMID: 26853433). However, these types of assays may not accurately represent biological function. Computational prediction tools and conservation analyses suggest that this variant may impact the protein, though this information is not predictive enough to determine pathogenicity. The p.Glu235Gln variant is located in a region of HNF1A that is essential to protein folding and stability, suggesting that this variant is in a functional domain and slightly supports pathogenicity (PMID: 26853433). In summary, while there is some suspicion for a pathogenic role, the clinical significance of this variant is uncertain. ACMG/AMP Criteria applied: PM2, PP3, PS3_Supporting, PM1_Supporting (Richards 2015). |
Molecular Genetics, |
RCV002051925 | SCV002318408 | likely benign | Maturity onset diabetes mellitus in young | criteria provided, single submitter | clinical testing | ||
Clinical Genomics, |
RCV002051925 | SCV002604995 | uncertain risk allele | Maturity onset diabetes mellitus in young | criteria provided, single submitter | research | Mutations in HNF1A gene can predispose to MODY3. It is associated with both micro and macrovascular complications of diabetes, especially cardiovascular complications. Associated with glucosuria. May respond well to sulfonylureas. However, more evidence is required to confer the association of this particular variant rs1353807357 with MODY3. |